554
Case report
Sclerosing osteomyelitis as a complication of pediatric femur fracture fixation James F. Mooney III Complications of flexible nailing of pediatric femur fractures include angular and rotational malunions, leg-length discrepancy, and, in rare instances, infection. To our knowledge, the development of a sclerosing type of chronic osteomyelitis, which appears most similar to chronic sclerosing osteomyelitis of Garre’, has not been reported as a complication of, or associated with, flexible nail fixation of a pediatric femur fracture. J Pediatr Orthop B 23:554–559 © 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins.
Journal of Pediatric Orthopaedics B 2014, 23:554–559
Introduction
(Fig. 3a and b). Further follow-up was recommended, but clinic appointments were not kept.
Flexible intramedullary fixation is used widely as a treatment modality for pediatric femur fractures [1]. Complications of flexible nailing, although relatively uncommon, include angular and rotational malunions, leg-length discrepancy, and, in rare instances, infection [2–5]. To our knowledge, the development of a sclerosing type of chronic osteomyelitis, which appears similar to chronic sclerosing osteomyelitis of Garre’ (CSO), has not been reported as a complication of, or associated with, surgical fixation of a pediatric femur fracture.
Case
In June 2006, a 7-year-old white male sustained a closed right femur fracture in a low-energy fall while skateboarding. Imaging and physical examination were consistent with a fracture of the femoral diaphysis. He underwent closed nailing of the fracture with titanium elastic nails (Fig. 1a and b; Synthes, Paoli, Pennsylvania, USA) and received standard perioperative intravenous antibiotic dosing, which included one dose in the operating room and two further postoperative doses at 8-h intervals. He was advanced to full weight-bearing at ∼ 4 weeks, and was then lost to follow-up ∼ 2 months postoperatively. He presented 3 years later for implant removal. He was fully active, had no complaints of pain, fever, or systemic signs of illness or infection. Radiographs (Fig. 2a and b) showed the fracture to be healed and the diaphysis remodeled. He underwent uncomplicated implant removal, and there were no visual or clinical abnormalities noted of the bone or the implants at the time of removal. He was seen for a postoperative checkup and radiography 2 weeks later. The images were read by a radiologist as normal postimplant changes. However, the orthopedic staff raised some concern with regard to unusual radiodensity within the femoral diaphysis 1060-152X © 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins
Keywords: femur fractures, osteomyelitis, flexible nailing Department of Orthopaedic Surgery, Medical University of South Carolina, Charleston, South Carolina, USA Correspondence to James F. Mooney, III, MD, Department of Orthopaedic Surgery, Medical University of South Carolina, Charleston, SC 29425, USA Tel: + 1 843 792 8765; e-mail: [email protected]
The patient presented to an outside hospital 3 years later with complaints of a 1-year history of persistent thigh pain. Imaging, including plain radiography, MRI (Fig. 4a–d), and computed tomography (CT) was performed. A CT-guided bone biopsy was performed, which was inconclusive. Subsequently, the patient was referred back to the original treating institution for management. The patient was thin with a complaint of mid-thigh pain. The pain was constant and not relieved by NSAIDs. His thigh was enlarged diffusely compared with the contralateral side and was tender to touch. He was afebrile and had no systemic signs of infection, although both erythrocyte sedimentation rate and C reactive protein level were elevated. After consultation between Pediatric Orthopedic and Musculoskeletal Radiology Departments, it was believed that these findings were most consistent with chronic osteomyelitis and possibly CSO. There was no evidence of sequestra on either plain radiographs or MRI images. Because of the radiographic and physical findings, it was felt that surgical management was indicated. Debridement of the canal, culturing of the bone, and antibiotic management were planned. At the time of surgery, a vent was made in the distal femur to decompress any increased pressure secondary to reaming. All attempts to pass a guidewire failed, and it became apparent that the medullary canal of the involved area of the diaphysis was essentially solid. It was necessary to saucerize the lateral cortex of the entire area of the involved diaphysis with a high-speed burr, and to use the burr to ‘recreate’ a central canal. A guidewire was then passed, and the canal was reamed under fluoroscopy and direct vision. Multiple tissue cultures and pathology samples were sent. Because of concern about the risk for fracture secondary to the extensive lateral defect, as well DOI: 10.1097/BPB.0000000000000103
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Sclerosing osteomyelitis Mooney 555
Fig. 1
(a)
(b) 05/20/2006 09:44:32
G
HT
IR
RI
05/20/2006 09:44:32
A HG I T S
A IS
kV: mAs EI=
PORTRAIT PFEM2
PORTRAIT PFEM2
kV: mAs EI=
(a) Postoperative anteroposterior radiograph after flexible nail fixation. (b) Postoperative lateral radiograph after flexible nail fixation.
Fig. 2
(a)
(b) 09/21/2009 14:47:09
09/21/2009 14:47:09
CRR PORTRAIT PFEM2
kV: mAs EI=269
CRR PORTRAIT PFEM2
kV: mAs EI=267
(a) Anteroposterior radiograph of the right femur before nail removal. (b) Lateral radiograph of the right femur before nail removal.
as the possibility of persistent infection, an antibiotic impregnated PMMA/Enders nail was created to provide support and for local antibiotic delivery (Fig. 5). The patient did well postoperatively, and reported significant decrease in his thigh pain. Because of the
unlocked nature of the PMMA/Enders nail, in combination with the large iatrogenic lateral defect, he was kept toe-touch weight bearing in this postoperative period. His C reactive protein levels returned to normal quickly, and all cultures were negative. He received 6 weeks of oral antibiotics as per Pediatric Infectious
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
556 Journal of Pediatric Orthopaedics B 2014, Vol 23 No 6
Fig. 3
(a)
(b) 10/26/2009 14:05:14
10/26/2009 14:05:14
EAC
EAC
PORTRAIT PFEM2
kV: mAs EI=250
PORTRAIT PFEM2
kV: mAs EI=244
(a) Anteroposterior radiograph of the right femur after nail removal. (b) Lateral radiograph of the right femur after nail removal.
Disease service recommendations. Pathology specimens showed a combination of viable and nonviable bone, but no evidence of active infection or inflammation. Approximately 1 month after initial debridement, the antibiotic nail was exchanged for a locked, trochanteric entry femoral nail (Fig. 6), and the canal was rereamed before placement of the new implant. At the most recent follow-up, he reported complete resolution of his thigh pain and has returned to full activities. There are no plans in place for intramedullary nail removal at this time.
CSO is very uncommon and clinically poorly understood. It has been reported in pediatric long bones, and a similar entity appears to involve the mandible in this age group [9]. Patients generally present with insidious onset of pain and swelling in the area of bony involvement, which is usually within the diaphyseal region. Symptoms may wax and wane over long periods of time. Radiographs show a variety of cystic and sclerotic changes, often with expansion of the area of bone involvement. Bone scans show increased uptake, and MRI changes are not well described in the literature.
Discussion
Differential diagnoses in patients with possible CSO include osteogenic sarcoma, osteoid osteoma, fibrous dysplasia, and Paget’s disease, among others. In addition, chronic osteomyelitis with an active infectious agent must be ruled out in each case. In light of this, it is recommended that patients with clinical and radiographic findings consistent with CSO undergo cultures and biopsy of the involved area to eliminate other diagnoses [9,10]. In some ways, CSO involving the long bones is a diagnosis of exclusion.
There are multiple published studies that have reviewed the complications of management of pediatric femur fractures with flexible nail fixation [1–5]. In addition, one group has presented complications associated with removal of these devices [6]. The vast majority of complications associated with flexible nail fixation involve angular or shortening issues, often secondary to use in patients with length-unstable fracture patterns or to use in older and/or heavier patients. Infectious complications are reported rarely and seem to be most commonly superficial infections, in some cases associated with nail prominence. There is one report of development of chronic osteomyelitis with sequestrum formation after flexible intramedullary fixation in a pediatric patient [7]. In addition, there is mention of a pediatric patient who developed deep infection and acute osteomyelitis after rigid locked nailing of an open femur fracture treated initially with an external fixator [8]. There are no reported cases, to our knowledge, of any type of sclerosing osteomyelitis after flexible nail fixation of the femur.
The most appropriate management of CSO is unclear. Some patients may respond adequately to NSAIDs and other pain medications after the diagnosis is confirmed, and in some patients the pain will subside slowly [9]. Pain and swelling may wax and wane over time. Data from the oral surgery and maxillofacial surgery literature suggest clinical and symptomatic improvements in patients with diffuse sclerosing osteomyelitis of the mandible with the use of bisphosphonates [11]. There are no published data that address this type of medical management in
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Sclerosing osteomyelitis Mooney 557
Fig. 4
(a)
(b) 03/11/2013 14:13:28
03/11/2013 14:12:24
R
R AP FEMUR - 2 VIEWS
SE:1
(c)
LATERAL FEMUR - 2 VIEWS
SE:1
(d) 15:35:02
15:48:5
L R P A
COR T1 RIGHT THIGH
IAL
SE:
COR IR RIGHT THIGH
IAL
SE:
(a) Anteroposterior radiograph of right femur at the time of presentation 3 years after nail removal. (b) Lateral radiograph of the right femur at time or presentation 3 years s/p nail removal. (c) T1 coronal image of femur at the time of presentation. (d) T2 coronal image of femur at the time of presentation.
pediatric orthopedic patients with long-bone involvement. Surgical management recommendations are varied, ranging from fenestration of the involved area to intramedullary reaming, and most aggressively to wide resection and reconstruction with either bone transport or free bone and tissue transfer [10,12–15]. Little beyond fenestration and symptomatic management has been reported in children with long-bone abnormalities, although there is one recent report on wide resection of an involved area of the tibia and reconstructive bone transport [10]. This case is unusual because of the association with a previously surgically managed femur fracture, as well as the aggressive method of management. As CSO had not been
reported after this type of injury and treatment, it was difficult to predict the results of the intraoperative biopsies and cultures, and the CT-guided biopsy obtained before referral did not provide any useful information. The patient was managed initially with the belief that this was most likely chronic osteomyelitis, and re-establishment of the medullary canal with supportive and high local dose antibiotic treatment with the PMMA/antibiotic nail was chosen as the best surgical treatment option. Antibiotic beads may have provided similar local antibacterial action, but would have lacked the structural support believed to be necessary because of the large lateral cortical saucerization defect necessary to reestablish the canal. The pathology and culture results were consistent with CSO because of the lack of significant inflammatory cells, and in light of these results, the antibiotic
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
558 Journal of Pediatric Orthopaedics B 2014, Vol 23 No 6
Fig. 5
Fig. 6 05/23/2013 14:25:47
T Femur with knee a.p. PFEM2
SE:1
06/27/2013 14:21:20
T Femur with knee a.p. PFEM2
SE:6
Anteroposterio image of the right femur with antibiotic PMMA/Enders nail in place.
Anteroposterio image of the right femur with a trochanteric entry nail in place at the most recent follow-up.
nail was exchanged for the locked nail. This implant should offer long-term stability to the area, act prophylactically to limit the risk of later fracture, as well as maintain the integrity of the femoral canal.
with titanium flexible nails. This has not been reported, to date, in the existing literature. The surgical management was successful in relieving the patient’s symptomatology, as well as in providing long-term stabilization of the femur. Although an aggressive technique, this twostage method seems an attractive alternative to other potential interventions, including bone resection requiring lengthy bone transport or microvascular tissue transfer and reconstruction.
Conclusion This patient appears to have developed a type of aseptic sclerosing osteomyelitis, or possibly CSO, after management of a pediatric diaphyseal femur fracture
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Sclerosing osteomyelitis Mooney 559
Acknowledgements Conflicts of interest
There are no conflicts of interest.
References 1 Ho CA, Skaggs DL, Tang CW, Kay RM. Use of flexible nails in pediatric femur fractures. J Pediatr Orthop 2006; 26:497–504. 2 Flynn JM, Hresko T, Reynolds RAK, Blasier RD, Davidson R, Kasser J. Titanium elastic nails for pediatric femur fractures: a multicenter study of early results with analysis of complications. J Pediatr Orthop 2001; 21:4–8. 3 Narayanan UG, Hyman JE, Wainwright AM, Rang M, Alman BA. Complications of elastic stable intramedullary nail fixation of pediatric femur fracture, and how to avoid them. J Pediatr Orthop 2004; 24:363–369. 4 Sink EL, Gralla J, Repine M. Complications of pediatric femur fractures treated with titanium nails. J Pediatr Orthop 2005; 25:577–580. 5 Wall EJ, Jain V, Vora V, Mehlman CT, Crawford AH. Complications of titanium and stainless steel elastic nail fixation of pediatric femur fractures. J Bone Joint Surg Am 2008; 90:1305–1313. 6 Levy JA, Podeszwa DA, Lebus G, Ho CA, Wimberly RL. Acute complications associated with removal of flexible intramedullary rods placed for pediatric femoral shaft fractures. J Pediatr Orthop 2013; 33:43–47. 7 Hobson SA, Livesley PJ. A case of osteomyelitis following flexible nailing of a femoral shaft fracture in an 11 year old. Injury 2004; 35:1327–1329.
8 9
10
11
12
13
14
15
Letts M, Jarvis J, Lawton L, Davidson D. Complications of rigid intramedullary rodding of femoral fractures in children. J Trauma 2002; 52:504–516. Segev E, Hayek S, Lokiec F, Ezra E, Issakov J, Wientroub S. Primary chronic sclerosing (Garre’s)osteomyelitis in children. J Pediatr Orthop B 2001; 10:360–364. Nikomarov D, Zaidman M, Katzman A, Keren Y, Eidelman M. New treatment option for sclerosing osteomyelitis of Garre. J Pediatr Orthop B 2013; 22:577–582. Kuijpers SC, de Jong E, Hamdy NA, van Merkesteyn JP. Initial results of the treatment of diffuse sclerosing osteomyelitis of the mandible with bisphosphonates. J Craniomaxillofac Surg 2011; 39:65–68. Bettin D, Roulaffs B, von Eiff C, Sciuk J. Reconstructive treatment of sclerosing osteomyelitis of the entire femur of 30 years’ duration with avoidance of segmental resection. Arch Orthop Trauma Surg 1999; 119:228–231. Lok V, Bal E, Sebik A, Aydinok H. Long-term results of treatment including creation of a gutter and muscle transposition for chroming sclerosing osteomyelitis. Acta Orthop Traumatol Turc 2009; 43:510–514. Pape HC, Krettek C, Maschek H, Regel G, Tscherne H. Fatal pulmonary embolization after reaming of the femoral medullary cavity in sclerosing osteomyelitis: a case report. J Orthop Trauma 1996; 10:429–432. Schwartz AJ, Jones NF, Seeger LL, Nelson SD, Eckardt JJ. Chronic sclerosing osteomyelitis treated with wide resection and vascularized fibular autograft: a case report. Am J Orthop 2010; 39:E28–E32.
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Case report
Sclerosing osteomyelitis as a complication of pediatric femur fracture fixation James F. Mooney III Complications of flexible nailing of pediatric femur fractures include angular and rotational malunions, leg-length discrepancy, and, in rare instances, infection. To our knowledge, the development of a sclerosing type of chronic osteomyelitis, which appears most similar to chronic sclerosing osteomyelitis of Garre’, has not been reported as a complication of, or associated with, flexible nail fixation of a pediatric femur fracture. J Pediatr Orthop B 23:554–559 © 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins.
Journal of Pediatric Orthopaedics B 2014, 23:554–559
Introduction
(Fig. 3a and b). Further follow-up was recommended, but clinic appointments were not kept.
Flexible intramedullary fixation is used widely as a treatment modality for pediatric femur fractures [1]. Complications of flexible nailing, although relatively uncommon, include angular and rotational malunions, leg-length discrepancy, and, in rare instances, infection [2–5]. To our knowledge, the development of a sclerosing type of chronic osteomyelitis, which appears similar to chronic sclerosing osteomyelitis of Garre’ (CSO), has not been reported as a complication of, or associated with, surgical fixation of a pediatric femur fracture.
Case
In June 2006, a 7-year-old white male sustained a closed right femur fracture in a low-energy fall while skateboarding. Imaging and physical examination were consistent with a fracture of the femoral diaphysis. He underwent closed nailing of the fracture with titanium elastic nails (Fig. 1a and b; Synthes, Paoli, Pennsylvania, USA) and received standard perioperative intravenous antibiotic dosing, which included one dose in the operating room and two further postoperative doses at 8-h intervals. He was advanced to full weight-bearing at ∼ 4 weeks, and was then lost to follow-up ∼ 2 months postoperatively. He presented 3 years later for implant removal. He was fully active, had no complaints of pain, fever, or systemic signs of illness or infection. Radiographs (Fig. 2a and b) showed the fracture to be healed and the diaphysis remodeled. He underwent uncomplicated implant removal, and there were no visual or clinical abnormalities noted of the bone or the implants at the time of removal. He was seen for a postoperative checkup and radiography 2 weeks later. The images were read by a radiologist as normal postimplant changes. However, the orthopedic staff raised some concern with regard to unusual radiodensity within the femoral diaphysis 1060-152X © 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins
Keywords: femur fractures, osteomyelitis, flexible nailing Department of Orthopaedic Surgery, Medical University of South Carolina, Charleston, South Carolina, USA Correspondence to James F. Mooney, III, MD, Department of Orthopaedic Surgery, Medical University of South Carolina, Charleston, SC 29425, USA Tel: + 1 843 792 8765; e-mail: [email protected]
The patient presented to an outside hospital 3 years later with complaints of a 1-year history of persistent thigh pain. Imaging, including plain radiography, MRI (Fig. 4a–d), and computed tomography (CT) was performed. A CT-guided bone biopsy was performed, which was inconclusive. Subsequently, the patient was referred back to the original treating institution for management. The patient was thin with a complaint of mid-thigh pain. The pain was constant and not relieved by NSAIDs. His thigh was enlarged diffusely compared with the contralateral side and was tender to touch. He was afebrile and had no systemic signs of infection, although both erythrocyte sedimentation rate and C reactive protein level were elevated. After consultation between Pediatric Orthopedic and Musculoskeletal Radiology Departments, it was believed that these findings were most consistent with chronic osteomyelitis and possibly CSO. There was no evidence of sequestra on either plain radiographs or MRI images. Because of the radiographic and physical findings, it was felt that surgical management was indicated. Debridement of the canal, culturing of the bone, and antibiotic management were planned. At the time of surgery, a vent was made in the distal femur to decompress any increased pressure secondary to reaming. All attempts to pass a guidewire failed, and it became apparent that the medullary canal of the involved area of the diaphysis was essentially solid. It was necessary to saucerize the lateral cortex of the entire area of the involved diaphysis with a high-speed burr, and to use the burr to ‘recreate’ a central canal. A guidewire was then passed, and the canal was reamed under fluoroscopy and direct vision. Multiple tissue cultures and pathology samples were sent. Because of concern about the risk for fracture secondary to the extensive lateral defect, as well DOI: 10.1097/BPB.0000000000000103
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Sclerosing osteomyelitis Mooney 555
Fig. 1
(a)
(b) 05/20/2006 09:44:32
G
HT
IR
RI
05/20/2006 09:44:32
A HG I T S
A IS
kV: mAs EI=
PORTRAIT PFEM2
PORTRAIT PFEM2
kV: mAs EI=
(a) Postoperative anteroposterior radiograph after flexible nail fixation. (b) Postoperative lateral radiograph after flexible nail fixation.
Fig. 2
(a)
(b) 09/21/2009 14:47:09
09/21/2009 14:47:09
CRR PORTRAIT PFEM2
kV: mAs EI=269
CRR PORTRAIT PFEM2
kV: mAs EI=267
(a) Anteroposterior radiograph of the right femur before nail removal. (b) Lateral radiograph of the right femur before nail removal.
as the possibility of persistent infection, an antibiotic impregnated PMMA/Enders nail was created to provide support and for local antibiotic delivery (Fig. 5). The patient did well postoperatively, and reported significant decrease in his thigh pain. Because of the
unlocked nature of the PMMA/Enders nail, in combination with the large iatrogenic lateral defect, he was kept toe-touch weight bearing in this postoperative period. His C reactive protein levels returned to normal quickly, and all cultures were negative. He received 6 weeks of oral antibiotics as per Pediatric Infectious
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
556 Journal of Pediatric Orthopaedics B 2014, Vol 23 No 6
Fig. 3
(a)
(b) 10/26/2009 14:05:14
10/26/2009 14:05:14
EAC
EAC
PORTRAIT PFEM2
kV: mAs EI=250
PORTRAIT PFEM2
kV: mAs EI=244
(a) Anteroposterior radiograph of the right femur after nail removal. (b) Lateral radiograph of the right femur after nail removal.
Disease service recommendations. Pathology specimens showed a combination of viable and nonviable bone, but no evidence of active infection or inflammation. Approximately 1 month after initial debridement, the antibiotic nail was exchanged for a locked, trochanteric entry femoral nail (Fig. 6), and the canal was rereamed before placement of the new implant. At the most recent follow-up, he reported complete resolution of his thigh pain and has returned to full activities. There are no plans in place for intramedullary nail removal at this time.
CSO is very uncommon and clinically poorly understood. It has been reported in pediatric long bones, and a similar entity appears to involve the mandible in this age group [9]. Patients generally present with insidious onset of pain and swelling in the area of bony involvement, which is usually within the diaphyseal region. Symptoms may wax and wane over long periods of time. Radiographs show a variety of cystic and sclerotic changes, often with expansion of the area of bone involvement. Bone scans show increased uptake, and MRI changes are not well described in the literature.
Discussion
Differential diagnoses in patients with possible CSO include osteogenic sarcoma, osteoid osteoma, fibrous dysplasia, and Paget’s disease, among others. In addition, chronic osteomyelitis with an active infectious agent must be ruled out in each case. In light of this, it is recommended that patients with clinical and radiographic findings consistent with CSO undergo cultures and biopsy of the involved area to eliminate other diagnoses [9,10]. In some ways, CSO involving the long bones is a diagnosis of exclusion.
There are multiple published studies that have reviewed the complications of management of pediatric femur fractures with flexible nail fixation [1–5]. In addition, one group has presented complications associated with removal of these devices [6]. The vast majority of complications associated with flexible nail fixation involve angular or shortening issues, often secondary to use in patients with length-unstable fracture patterns or to use in older and/or heavier patients. Infectious complications are reported rarely and seem to be most commonly superficial infections, in some cases associated with nail prominence. There is one report of development of chronic osteomyelitis with sequestrum formation after flexible intramedullary fixation in a pediatric patient [7]. In addition, there is mention of a pediatric patient who developed deep infection and acute osteomyelitis after rigid locked nailing of an open femur fracture treated initially with an external fixator [8]. There are no reported cases, to our knowledge, of any type of sclerosing osteomyelitis after flexible nail fixation of the femur.
The most appropriate management of CSO is unclear. Some patients may respond adequately to NSAIDs and other pain medications after the diagnosis is confirmed, and in some patients the pain will subside slowly [9]. Pain and swelling may wax and wane over time. Data from the oral surgery and maxillofacial surgery literature suggest clinical and symptomatic improvements in patients with diffuse sclerosing osteomyelitis of the mandible with the use of bisphosphonates [11]. There are no published data that address this type of medical management in
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Sclerosing osteomyelitis Mooney 557
Fig. 4
(a)
(b) 03/11/2013 14:13:28
03/11/2013 14:12:24
R
R AP FEMUR - 2 VIEWS
SE:1
(c)
LATERAL FEMUR - 2 VIEWS
SE:1
(d) 15:35:02
15:48:5
L R P A
COR T1 RIGHT THIGH
IAL
SE:
COR IR RIGHT THIGH
IAL
SE:
(a) Anteroposterior radiograph of right femur at the time of presentation 3 years after nail removal. (b) Lateral radiograph of the right femur at time or presentation 3 years s/p nail removal. (c) T1 coronal image of femur at the time of presentation. (d) T2 coronal image of femur at the time of presentation.
pediatric orthopedic patients with long-bone involvement. Surgical management recommendations are varied, ranging from fenestration of the involved area to intramedullary reaming, and most aggressively to wide resection and reconstruction with either bone transport or free bone and tissue transfer [10,12–15]. Little beyond fenestration and symptomatic management has been reported in children with long-bone abnormalities, although there is one recent report on wide resection of an involved area of the tibia and reconstructive bone transport [10]. This case is unusual because of the association with a previously surgically managed femur fracture, as well as the aggressive method of management. As CSO had not been
reported after this type of injury and treatment, it was difficult to predict the results of the intraoperative biopsies and cultures, and the CT-guided biopsy obtained before referral did not provide any useful information. The patient was managed initially with the belief that this was most likely chronic osteomyelitis, and re-establishment of the medullary canal with supportive and high local dose antibiotic treatment with the PMMA/antibiotic nail was chosen as the best surgical treatment option. Antibiotic beads may have provided similar local antibacterial action, but would have lacked the structural support believed to be necessary because of the large lateral cortical saucerization defect necessary to reestablish the canal. The pathology and culture results were consistent with CSO because of the lack of significant inflammatory cells, and in light of these results, the antibiotic
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
558 Journal of Pediatric Orthopaedics B 2014, Vol 23 No 6
Fig. 5
Fig. 6 05/23/2013 14:25:47
T Femur with knee a.p. PFEM2
SE:1
06/27/2013 14:21:20
T Femur with knee a.p. PFEM2
SE:6
Anteroposterio image of the right femur with antibiotic PMMA/Enders nail in place.
Anteroposterio image of the right femur with a trochanteric entry nail in place at the most recent follow-up.
nail was exchanged for the locked nail. This implant should offer long-term stability to the area, act prophylactically to limit the risk of later fracture, as well as maintain the integrity of the femoral canal.
with titanium flexible nails. This has not been reported, to date, in the existing literature. The surgical management was successful in relieving the patient’s symptomatology, as well as in providing long-term stabilization of the femur. Although an aggressive technique, this twostage method seems an attractive alternative to other potential interventions, including bone resection requiring lengthy bone transport or microvascular tissue transfer and reconstruction.
Conclusion This patient appears to have developed a type of aseptic sclerosing osteomyelitis, or possibly CSO, after management of a pediatric diaphyseal femur fracture
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Sclerosing osteomyelitis Mooney 559
Acknowledgements Conflicts of interest
There are no conflicts of interest.
References 1 Ho CA, Skaggs DL, Tang CW, Kay RM. Use of flexible nails in pediatric femur fractures. J Pediatr Orthop 2006; 26:497–504. 2 Flynn JM, Hresko T, Reynolds RAK, Blasier RD, Davidson R, Kasser J. Titanium elastic nails for pediatric femur fractures: a multicenter study of early results with analysis of complications. J Pediatr Orthop 2001; 21:4–8. 3 Narayanan UG, Hyman JE, Wainwright AM, Rang M, Alman BA. Complications of elastic stable intramedullary nail fixation of pediatric femur fracture, and how to avoid them. J Pediatr Orthop 2004; 24:363–369. 4 Sink EL, Gralla J, Repine M. Complications of pediatric femur fractures treated with titanium nails. J Pediatr Orthop 2005; 25:577–580. 5 Wall EJ, Jain V, Vora V, Mehlman CT, Crawford AH. Complications of titanium and stainless steel elastic nail fixation of pediatric femur fractures. J Bone Joint Surg Am 2008; 90:1305–1313. 6 Levy JA, Podeszwa DA, Lebus G, Ho CA, Wimberly RL. Acute complications associated with removal of flexible intramedullary rods placed for pediatric femoral shaft fractures. J Pediatr Orthop 2013; 33:43–47. 7 Hobson SA, Livesley PJ. A case of osteomyelitis following flexible nailing of a femoral shaft fracture in an 11 year old. Injury 2004; 35:1327–1329.
8 9
10
11
12
13
14
15
Letts M, Jarvis J, Lawton L, Davidson D. Complications of rigid intramedullary rodding of femoral fractures in children. J Trauma 2002; 52:504–516. Segev E, Hayek S, Lokiec F, Ezra E, Issakov J, Wientroub S. Primary chronic sclerosing (Garre’s)osteomyelitis in children. J Pediatr Orthop B 2001; 10:360–364. Nikomarov D, Zaidman M, Katzman A, Keren Y, Eidelman M. New treatment option for sclerosing osteomyelitis of Garre. J Pediatr Orthop B 2013; 22:577–582. Kuijpers SC, de Jong E, Hamdy NA, van Merkesteyn JP. Initial results of the treatment of diffuse sclerosing osteomyelitis of the mandible with bisphosphonates. J Craniomaxillofac Surg 2011; 39:65–68. Bettin D, Roulaffs B, von Eiff C, Sciuk J. Reconstructive treatment of sclerosing osteomyelitis of the entire femur of 30 years’ duration with avoidance of segmental resection. Arch Orthop Trauma Surg 1999; 119:228–231. Lok V, Bal E, Sebik A, Aydinok H. Long-term results of treatment including creation of a gutter and muscle transposition for chroming sclerosing osteomyelitis. Acta Orthop Traumatol Turc 2009; 43:510–514. Pape HC, Krettek C, Maschek H, Regel G, Tscherne H. Fatal pulmonary embolization after reaming of the femoral medullary cavity in sclerosing osteomyelitis: a case report. J Orthop Trauma 1996; 10:429–432. Schwartz AJ, Jones NF, Seeger LL, Nelson SD, Eckardt JJ. Chronic sclerosing osteomyelitis treated with wide resection and vascularized fibular autograft: a case report. Am J Orthop 2010; 39:E28–E32.
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
